A Rasch Analysis of the Charcot-Marie-Tooth Neuropathy Score (CMTNS) in a Cohort of Charcot-Marie-Tooth Type 1A Patients - Archive ouverte HAL Accéder directement au contenu
Article Dans Une Revue PLoS ONE Année : 2017

A Rasch Analysis of the Charcot-Marie-Tooth Neuropathy Score (CMTNS) in a Cohort of Charcot-Marie-Tooth Type 1A Patients

Mickaël Guedj
  • Fonction : Auteur
Viviane Bertrand
  • Fonction : Auteur
Julie Foucquier
  • Fonction : Auteur
Elisabeth Jouve
  • Fonction : Auteur
Niall Murphy
  • Fonction : Auteur
Olivier Blin
Daniel Cohen
  • Fonction : Auteur
  • PersonId : 760130
  • IdRef : 032639805

Résumé

The Charcot-Marie-Tooth Neuropathy Score (CMTNS) was developed as a main efficacy endpoint for application in clinical trials of Charcot-Marie-Tooth disease type 1A (CMT1A). However, the sensitivity of the CMTNS for measuring disease severity and progression in CMT1A patients has been questioned. Here, we applied a Rasch analysis in a French cohort of patients to evaluate the psychometrical properties of the CMTNS. Overall, our analysis supports the validity of the CMTNS for application to CMT1A patients though with some limitations such as certain items of the CMTNS being more suitable for moderate to severe forms of the disease, and some items being disordered. We suggest that additional items and/or categories be considered to better assess mild-to-moderate patients.

Domaines

Neurosciences

Dates et versions

hal-03598218 , version 1 (04-03-2022)

Identifiants

Citer

Wenjia Wang, Mickaël Guedj, Viviane Bertrand, Julie Foucquier, Elisabeth Jouve, et al.. A Rasch Analysis of the Charcot-Marie-Tooth Neuropathy Score (CMTNS) in a Cohort of Charcot-Marie-Tooth Type 1A Patients. PLoS ONE, 2017, 12 (1), pp.e0169878. ⟨10.1371/journal.pone.0169878⟩. ⟨hal-03598218⟩
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